{"title":"儿童免疫球蛋白 A 血管炎的胃肠道表现和发病机制。","authors":"Seiichi Kato, Benjamin D Gold, Ayumu Kato","doi":"10.3389/fped.2024.1459394","DOIUrl":null,"url":null,"abstract":"<p><p>Immunoglobulin A vasculitis (IgAV), previously known as Henoch-Schönlein purpura, is the most common form of systemic vasculitis in childhood. The primary organs involved are the skin, gastrointestinal (GI) tract, joints, and kidneys. The spectrum of GI involvement in IgAV ranges from being mild and self-limited to severe manifestations often requiring surgical intervention. Galactose-deficient IgA1 on the immunoglobulin hinge region and its immune complexes are thought to play a central pathogenetic role in IgAV, however, an association between such molecules and specific GI mucosal damage remains unclear. GI endoscopy (both upper and lower) shows a variety of mucosal findings, many of which are not specific for IgAV. In upper GI endoscopy, however, the mucosal features can be diagnostic when found localized in the more distal part of upper GI tract (second and/or third parts of the duodenum). Abdominal computed tomography and capsule endoscopy have demonstrated that the small intestine is most commonly involved in IgAV. The GI mucosal involvement when evaluated microscopically shows IgA deposition which is histologically diagnostic. Conversely, leukocytoclastic vasculitis is less useful. Since the 1960s, cases of duodenojejunitis, in which IgAV was suspected but evident purpura was not dermatologically present, have often been labeled as \"idiopathic\". In a pediatric case series, IgA enteropathy, without dermatological manifestations (i.e., purpura), was reported to have similar symptoms, as well as endoscopic characteristics and immunohistological findings as in IgAV. Subsequently, several case reports provide additional supportive evidence that IgA enteropathy must be a variant of IgAV. Thus, the immunologically driven auto-immune vasculitis results in the symptom complex dependent on the organ system involved, and the subsequent clinical features which are manifested. Present classification criteria are useful and universally available for diagnosing IgAV. However, based upon current knowledge including IgA enteropathy, minor modification of the IgAV criteria is proposed in the review.</p>","PeriodicalId":12637,"journal":{"name":"Frontiers in Pediatrics","volume":"12 ","pages":"1459394"},"PeriodicalIF":2.1000,"publicationDate":"2024-10-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11532042/pdf/","citationCount":"0","resultStr":"{\"title\":\"Gastrointestinal manifestations and pathogenesis in childhood immunoglobulin A vasculitis.\",\"authors\":\"Seiichi Kato, Benjamin D Gold, Ayumu Kato\",\"doi\":\"10.3389/fped.2024.1459394\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Immunoglobulin A vasculitis (IgAV), previously known as Henoch-Schönlein purpura, is the most common form of systemic vasculitis in childhood. The primary organs involved are the skin, gastrointestinal (GI) tract, joints, and kidneys. The spectrum of GI involvement in IgAV ranges from being mild and self-limited to severe manifestations often requiring surgical intervention. Galactose-deficient IgA1 on the immunoglobulin hinge region and its immune complexes are thought to play a central pathogenetic role in IgAV, however, an association between such molecules and specific GI mucosal damage remains unclear. GI endoscopy (both upper and lower) shows a variety of mucosal findings, many of which are not specific for IgAV. In upper GI endoscopy, however, the mucosal features can be diagnostic when found localized in the more distal part of upper GI tract (second and/or third parts of the duodenum). Abdominal computed tomography and capsule endoscopy have demonstrated that the small intestine is most commonly involved in IgAV. The GI mucosal involvement when evaluated microscopically shows IgA deposition which is histologically diagnostic. Conversely, leukocytoclastic vasculitis is less useful. 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引用次数: 0
摘要
免疫球蛋白 A 型血管炎(IgAV)以前被称为过敏性紫癜(Henoch-Schönlein purpura),是儿童时期最常见的全身性血管炎。主要受累器官是皮肤、胃肠道(GI)、关节和肾脏。IgAV 的胃肠道受累范围从轻微的自限性表现到通常需要手术干预的严重表现不等。免疫球蛋白铰链区的半乳糖缺失 IgA1 及其免疫复合物被认为在 IgAV 中起着核心的致病作用,但这些分子与特定的消化道粘膜损伤之间的联系仍不清楚。消化道内窥镜检查(包括上消化道内窥镜和下消化道内窥镜)可发现多种粘膜病变,其中许多病变对 IgAV 并无特异性。不过,在上消化道内镜检查中,如果发现上消化道较远的局部(十二指肠的第二和/或第三部分)有粘膜特征,则可以诊断。腹部计算机断层扫描和胶囊内镜检查表明,IgAV 最常累及小肠。消化道粘膜受累在显微镜下可看到 IgA 沉积,这在组织学上具有诊断意义。相反,白细胞凝集性血管炎的诊断价值较低。自 20 世纪 60 年代以来,怀疑有 IgAV 但皮肤学上没有明显紫癜的十二指肠空肠炎病例常常被称为 "特发性"。据报道,在一个儿科病例系列中,无皮肤表现(即紫癜)的 IgA 肠病具有与 IgAV 相似的症状、内镜特征和免疫组织学发现。随后,一些病例报告提供了更多支持性证据,证明 IgA 肠病一定是 IgAV 的一种变异型。因此,免疫驱动的自身免疫性血管炎导致的综合症状取决于所涉及的器官系统以及随后表现出的临床特征。目前的分类标准对诊断 IgAV 非常有用,而且普遍适用。然而,根据目前的知识,包括 IgA 肠病,本综述建议对 IgAV 标准稍作修改。
Gastrointestinal manifestations and pathogenesis in childhood immunoglobulin A vasculitis.
Immunoglobulin A vasculitis (IgAV), previously known as Henoch-Schönlein purpura, is the most common form of systemic vasculitis in childhood. The primary organs involved are the skin, gastrointestinal (GI) tract, joints, and kidneys. The spectrum of GI involvement in IgAV ranges from being mild and self-limited to severe manifestations often requiring surgical intervention. Galactose-deficient IgA1 on the immunoglobulin hinge region and its immune complexes are thought to play a central pathogenetic role in IgAV, however, an association between such molecules and specific GI mucosal damage remains unclear. GI endoscopy (both upper and lower) shows a variety of mucosal findings, many of which are not specific for IgAV. In upper GI endoscopy, however, the mucosal features can be diagnostic when found localized in the more distal part of upper GI tract (second and/or third parts of the duodenum). Abdominal computed tomography and capsule endoscopy have demonstrated that the small intestine is most commonly involved in IgAV. The GI mucosal involvement when evaluated microscopically shows IgA deposition which is histologically diagnostic. Conversely, leukocytoclastic vasculitis is less useful. Since the 1960s, cases of duodenojejunitis, in which IgAV was suspected but evident purpura was not dermatologically present, have often been labeled as "idiopathic". In a pediatric case series, IgA enteropathy, without dermatological manifestations (i.e., purpura), was reported to have similar symptoms, as well as endoscopic characteristics and immunohistological findings as in IgAV. Subsequently, several case reports provide additional supportive evidence that IgA enteropathy must be a variant of IgAV. Thus, the immunologically driven auto-immune vasculitis results in the symptom complex dependent on the organ system involved, and the subsequent clinical features which are manifested. Present classification criteria are useful and universally available for diagnosing IgAV. However, based upon current knowledge including IgA enteropathy, minor modification of the IgAV criteria is proposed in the review.
期刊介绍:
Frontiers in Pediatrics (Impact Factor 2.33) publishes rigorously peer-reviewed research broadly across the field, from basic to clinical research that meets ongoing challenges in pediatric patient care and child health. Field Chief Editors Arjan Te Pas at Leiden University and Michael L. Moritz at the Children''s Hospital of Pittsburgh are supported by an outstanding Editorial Board of international experts. This multidisciplinary open-access journal is at the forefront of disseminating and communicating scientific knowledge and impactful discoveries to researchers, academics, clinicians and the public worldwide.
Frontiers in Pediatrics also features Research Topics, Frontiers special theme-focused issues managed by Guest Associate Editors, addressing important areas in pediatrics. In this fashion, Frontiers serves as an outlet to publish the broadest aspects of pediatrics in both basic and clinical research, including high-quality reviews, case reports, editorials and commentaries related to all aspects of pediatrics.